Literature DB >> 29531005

A Novel Frameshift Mutation (c.5387_5388insTT) in NIPBL in Cornelia de Lange Syndrome with Severe Phenotype.

Min Jae Kang1, Soo Min Ahn2, Il Tae Hwang3.   

Abstract

Cornelia de Lange syndrome (CdLS) is a developmental disorder which is characterized by typical facial features, upper extremity malformations, and growth and cognitive delays. The genes involved in CdLS encode the cohesin complex and its associated proteins; and NIPBL mutations, which account for half of the cases, result in severe CdLS phenotypes. We describe a girl with CdLS, presenting with typical facial dysmorphism, cleft palate, hypertrichosis, upper limb hypertonicity, flexion contracture of elbows, micromelia, bilateral hearing loss, gastroesophageal reflux, and severe pyloric stenosis. We detected a heterozygous frameshift mutation in NIPBL (c.5387_5388ins(TT), p.Leu1796Phefs*8) which is a novel mutation.
© 2018 by the Association of Clinical Scientists, Inc.

Entities:  

Keywords:  Cornelia de Lange syndrome; Frameshift mutation; Korean; NIPBL

Mesh:

Substances:

Year:  2018        PMID: 29531005

Source DB:  PubMed          Journal:  Ann Clin Lab Sci        ISSN: 0091-7370            Impact factor:   1.256


  2 in total

1.  Case Report: Prenatal Whole-Exome Sequencing to Identify a Novel Heterozygous Synonymous Variant in NIPBL in a Fetus With Cornelia de Lange Syndrome.

Authors:  Fengchang Qiao; Cuiping Zhang; Yan Wang; Gang Liu; Binbin Shao; Ping Hu; Zhengfeng Xu
Journal:  Front Genet       Date:  2021-02-09       Impact factor: 4.599

2.  Delineation of phenotypes and genotypes related to cohesin structural protein RAD21.

Authors:  Lianne C Krab; Iñigo Marcos-Alcalde; Melissa Assaf; Meena Balasubramanian; Janne Bayer Andersen; Anne-Marie Bisgaard; David R Fitzpatrick; Sanna Gudmundsson; Sylvia A Huisman; Tugba Kalayci; Saskia M Maas; Francisco Martinez; Shane McKee; Leonie A Menke; Paul A Mulder; Oliver D Murch; Michael Parker; Juan Pie; Feliciano J Ramos; Claudine Rieubland; Jill A Rosenfeld Mokry; Emanuela Scarano; Marwan Shinawi; Paulino Gómez-Puertas; Zeynep Tümer; Raoul C Hennekam
Journal:  Hum Genet       Date:  2020-03-19       Impact factor: 4.132

  2 in total

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